Efficacy of Intralesional Aqueous Bleomycin in Treatment of Pediatric Lymphatic Malformations

Abstract Introduction: Lymphatic malformations (LMs) are an aberrant developmental anomaly of dysplastic sequestrated lymphatic vessels. Though surgical excision has been the conventional treatment modality, it has been associated with injury to adjacent structures, recurrences, and poor cosmesis. Intralesional bleomycin has shown a promising role in the treatment of LMs, and we present our experience with aqueous intralesional bleomycin in the pediatric population including infants in terms of its efficacy, complications, and parental satisfaction. Materials and Methods: Children with clinically and ultrasound-proven macrocystic non-visceral LMs received aqueous bleomycin intralesionally under ultrasound guidance at 0.5 IU/kg/dose at monthly intervals and not exceeding five doses. Response to therapy and complications were monitored and regression graded as excellent (total disappearance), good (>50% reduction), and poor (<50% reduction). Results: Our study included 26 children with a median age of 24 months (range 2–168) and a median weight of 12 kg (range 4–38). The most common site was the neck (37%). Duration of follow-up was 6 years (range 1–7). Response was excellent in 23/26 (88.4%) patients, good in 3/26 (11.5%), and poor in none (0%). Nine (34.6%) patients showed complete response with a single injection. None (0%) developed recurrence. Three (11.5%) children had minor complications such as fever and mild hypopigmentation. None developed any major complications. Parental satisfaction was excellent in 24/26 (92.3%) and good in 2/26 (7.7%) patients. Conclusion: Intralesional aqueous bleomycin sclerotherapy is a safe non-surgical intervention for macrocystic pediatric LMs with reasonably good outcomes and even better parental satisfaction.

[1]  Z. Neyaz,et al.  Aqueous intralesional bleomycin sclerotherapy in lymphatic malformation: Our experience with children and adult , 2017, National journal of maxillofacial surgery.

[2]  J. Reekers,et al.  Intralesional Bleomycin Injections for Vascular Malformations: A Systematic Review and Meta-Analysis , 2016, Plastic and reconstructive surgery.

[3]  Gang Chen,et al.  Bleomycin induces endothelial mesenchymal transition through activation of mTOR pathway: a possible mechanism contributing to the sclerotherapy of venous malformations , 2013, British journal of pharmacology.

[4]  D. Ballah,et al.  Percutaneous sclerotherapy in neonatal and infant head and neck lymphatic malformations: a single center experience. , 2011, Journal of pediatric surgery.

[5]  R. Azizkhan,et al.  Noninterventional treatment of selected head and neck lymphatic malformations. , 2008, Journal of pediatric surgery.

[6]  G. Wirth,et al.  Slow-Flow Vascular Malformations , 2007, Clinical pediatrics.

[7]  R. Kanojia,et al.  Non surgical management of cystic lymphangioma , 2006, Indian journal of otolaryngology and head and neck surgery : official publication of the Association of Otolaryngologists of India.

[8]  J. Deveikis Percutaneous ethanol sclerotherapy for vascular malformations in the head and neck. , 2005, Archives of facial plastic surgery.

[9]  Richard J. H. Smith,et al.  New Treatment Options for Lymphangioma in Infants and Children , 2002, The Annals of otology, rhinology, and laryngology.

[10]  B. Charabi Cystic Hygroma of the Head and Neck - A Long-term Follow-up of 44 Cases , 2000, Acta oto-laryngologica. Supplementum.

[11]  C. Kullendorff Efficacy of bleomycin treatment for symptomatic hemangiomas in children , 1997, Pediatric Surgery International.

[12]  H. Mocan,et al.  A new treatment with bleomycin for complicated cutaneous hemangioma in children. , 1997, European journal of pediatric surgery : official journal of Austrian Association of Pediatric Surgery ... [et al] = Zeitschrift fur Kinderchirurgie.

[13]  A. Barker,et al.  Bleomycin therapy for cystic hygroma. , 1995, Journal of pediatric surgery.

[14]  M. Fukuzawa,et al.  Injection of bleomycin as a primary therapy of cystic lymphangioma. , 1992, Journal of pediatric surgery.

[15]  N. Tsuruga,et al.  Bleomycin treatment for cystic hygroma in children. , 1977, Nihon geka hokan. Archiv fur japanische Chirurgie.

[16]  M. Hashida,et al.  Sclerosing therapy with bleomycin emulsion for lymphangioma in children , 2004, Pediatric Surgery International.

[17]  K. E. Newhouse Goodman and Gilman's The Pharmacological Basis of Therapeutics , 1986, The Yale Journal of Biology and Medicine.

[18]  J. Chappuis [Current aspects of cystic lymphangioma in the neck]. , 1994, Archives de pediatrie : organe officiel de la Societe francaise de pediatrie.